Prenatal diagnosis of cystic adenomatoid malformation of one fetus in a twin pregnancy: an unusual presentation

Bronchopulmonary‐foregut malformations:A continuum of paracrine hamartomas?

“…The compound lesions show overlapping clinical manifestations, despite morphologic distinctiveness of the participating hamartomas [Budorick et al, 1992;Demos and Teresi, 1975;Moerman et al, 1992;Zangwill and Stocker, 1993;Dolkart et al, 1992], while the four types have clinical and radiological/ sonographic signs and symptoms that allow diagnostic differentiation without histologic studies. Most of these malformations are solitary, nonhereditary, and with normal chromosomes; yet they have been reported with trisomies 18 and 21 [Moerman et al, 1992;Levkoff et al, 1964;Bromley et al, 1995] and with 47,XXY [Revillon et al, 1993], in monochorionic diamnionic twins [Moerman et al, 1992;Rebarber and Mohan, 1992;Harper, 1992;Sandoz, 1907;Bromley et al, 1995], with monogenic traits such as urogenital adysplasia [Moerman et al, 1992] and brachymorphismonychodysplasia-dysphalangism syndrome [Verloes et al, 1993], and with other hamartomas, Jadassohn sebaceous nevus [Sweeney et al, 1994], nephromegaly [Graham et al, 1987;Scully et al, 1985;Weinberg and Zumwalt, 1977], and renal cysts [Graham et al, 1987;Conway, 1951;Roloff et al, 1971]. In Proteus and basalcell nevus syndromes there have also been pulmonary hamartomas resembling BPFM [Cohen, 1993;Totten, 1980].…”

“…There are several reports of familial BPFM [MacRae, 1947;Sandoz, 1907;Krous and Sexauer, 1981]. CCAML is the most frequently diagnosed pulmonary hamartoma in utero [Heydanus et al, 1993;Petit et al, 1987;Boulot et al, 1991;Rebarber and Mohan, 1992;Revillon et al, 1993;Sakala et al, 1994;Kuller et al, 1992], and may be the most common postnatal lesion [Coran and Drongowski, 1994;Bailey et al, 1990;Cloutier et al, 1993;Richards et al, 1992;Revillon et al, 1993]. On histologic examination, three types of CCAML have been delineated [Stocker et al, 1977], and recently, based on the site of the defect in the tracheobronchial tree, Stocker [1994] expanded his classification to five types, with types 0 and 4 being exceedingly rare.…”